ArticleThe American journal of pathology2015
Multiple requirements of the focal dermal hypoplasia gene porcupine during ocular morphogenesis.
Article in The American journal of pathology, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 20 papers.
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Who cites it
20 citing papers in PubMed, 28 citations in OpenAlex.
- Direct Comparison of Constitutive Rax-Cre Transgenic Drivers That Activate in the Mouse Embryonic Eye Field.Investigative ophthalmology & visual science · 2026Article
- Direct comparison of constitutive Rax-Cre transgenic drivers that activate in the mouse embryonic eye field.bioRxiv : the preprint server for biology · 2025Article
- Fine-tuning of Wnt signaling by RNA surveillance factor Smg5 in the mouse craniofacial development.iScience · 2025Article
- Loss of Cdc42 causes abnormal optic cup morphogenesis and microphthalmia in mouse.bioRxiv : the preprint server for biology · 2024Article
- Loss of Cdc42 causes abnormal optic cup morphogenesis and microphthalmia in mouse.Frontiers in cellular neuroscience · 2024Article
- The role of Evi/Wntless in exporting Wnt proteins.Development (Cambridge, England) · 2023Review
- Optic cup morphogenesis across species and related inborn human eye defects.Development (Cambridge, England) · 2023Review
- Porcn is essential for growth and invagination of the mammalian optic cup.Frontiers in cell and developmental biology · 2022Article
- Selective Requirements for Vascular Endothelial Cells and Circulating Factors in the Regulation of Retinal Neurogenesis.Frontiers in cell and developmental biology · 2021Article
- Nf2 fine-tunes proliferation and tissue alignment during closure of the optic fissure in the embryonic mouse eye.Human molecular genetics · 2020Article
- In Vivo Analysis of Optic Fissure Fusion in Zebrafish: Pioneer Cells, Basal Lamina, Hyaloid Vessels, and How Fissure Fusion is Affected by BMP.International journal of molecular sciences · 2020Article
- MicroRNA-124-3p suppresses mouse lip mesenchymal cell proliferation through the regulation of genes associated with cleft lip in the mouse.BMC genomics · 2019Article
- Transcriptomic analysis of differential gene expression during chick periocular neural crest differentiation into corneal cells.Developmental dynamics : an official publication of the American Association of Anatomists · 2019Article
- The molecular anatomy of mammalian upper lip and primary palate fusion at single cell resolution.Development (Cambridge, England) · 2019Article
- Morphogenesis and axis specification occur in parallel during optic cup and optic fissure formation, differentially modulated by BMP and Wnt.Open biology · 2019Article
- Wnt signaling in orofacial clefts: crosstalk, pathogenesis and models.Disease models & mechanisms · 2019Review
- TGFβ-facilitated optic fissure fusion and the role of bone morphogenetic protein antagonism.Open biology · 2018Article
- Magnetic resonance imaging study of eye congenital birth defects in mouse model.Molecular vision · 2017Article
- Loss of Axin2 Causes Ocular Defects During Mouse Eye Development.Investigative ophthalmology & visual science · 2016Article
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Authors and funding
6 authors at 2 institutions in 1 country.
Funding
Abstract
Wnt glycoproteins control key processes during development and disease by activating various downstream pathways. Wnt secretion requires post-translational modification mediated by the O-acyltransferase encoded by the Drosophila porcupine homolog gene (PORCN). In humans, PORCN mutations cause focal dermal hypoplasia (FDH, or Goltz syndrome), an X-linked dominant multisystem birth defect that is frequently accompanied by ocular abnormalities such as coloboma, microphthalmia, or even anophthalmia. Although genetic ablation of Porcn in mouse has provided insight into the etiology of defects caused by ectomesodermal dysplasia in FDH, the requirement for Porcn and the actual Wnt ligands during eye development have been unknown. In this study, Porcn hemizygosity occasionally caused ocular defects reminiscent of FDH. Conditional inactivation of Porcn in periocular mesenchyme led to defects in mid- and hindbrain and in craniofacial development, but was insufficient to cause ocular abnormalities. However, a combination of conditional Porcn depletion in optic vesicle neuroectoderm, lens, and neural crest-derived periocular mesenchyme induced severe eye abnormalities with high penetrance. In particular, we observed coloboma, transdifferentiation of the dorsal and ventral retinal pigment epithelium, defective optic cup periphery, and closure defects of the eyelid, as well as defective corneal morphogenesis. Thus, Porcn is required in both extraocular and neuroectodermal tissues to regulate distinct Wnt-dependent processes during morphogenesis of the posterior and anterior segments of the eye.
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